OSTEOID-OSTEOMA Physical

examination
toe.

PRODUCING in the distal of the There toe.

as

PREMATURE

FUSION

121 phalanx closure of the big toe and of the epiphysis of

revealed

tenderness

to palpation warmth, of the with

1973, 22,
toe

were that

that it

it occurred produced a

in the distal premature

phalanx
nail
was

of the big or increased enlargement normal and

There sweating. of the

made

was no increased The toe

on

hypertrophy was normal.

were interpreted

bed,
some

motion

as compared
February

the opposite nidus 1973, phalanx was

Roentgenograms

being (Fig. definite toe The

within 1). (Fig. nidus 2).

limits, closure

although made

in retrospect on September

a small 14,

visible a

adjacent to it. This is the first reported case the epiphysis of a big toe associated

of premature closure with osteoid-osteoma

Roentgenograms Laboratory excision diagnosis

revealed

of the epiphysis

of the distal

of the big 12
,

An en bloc

studies were within normal of the lesion was performed was osteoid-osteoma (Fig.

limits. on October 3). The

1973.
had

pathological

patient

and is only the third reported case of premature epiphyseal closure associated with osteoid-osteoma. The normal age for closure of the epiphysis of the distal phalanx in a male is seventeen to eighteen years In our patient the epiphysis of
.

complete

relief

of pain.

the Discussion

opposite big The specific with

toe had not closed. cause of the premature osteoid-osteoma is not

epiphyseal known.

closure In this

associated case are typical of an had the typical of the lesion and aspects of the lesion particular because lesion increased

The osteoid-osteoma roentgenographic relief

findings

in

this

in that the patient picture, with progression by aspirin. Two unusual

case we think that the premature closure occurred of local damage to the epiphyseal plate by the directly, in conjunction and with regional the secondary osteoporosis. effects of vascularity

of pain

References
1. DE
2. WET, I. S.: Osteoid Osteomata. Review of the Literature with a Report of Five Cases. South FREIBERGER, R. H. ; LOITMAN, B. S.; HELPERN, NHLTON; and THOMPSON, T. C. : Osteoid Osteoma.
, 82: 1959. GIUSTRA, P. E. , and FREIBERGER, R. H. : Severe Growth Disturbance with Osteoid Osteoma. A Report of Two Cases Involving the Femoral Neck. Radiology, 96: 285-288, 1970. GRAY, HENRY: Anatomy of the Human Body. Ed. 33, edited by D. V. Davies and F. Davies. London, Longmans, 1962. JACKSON, I. J. : Osteoid Osteoma of the Lamina and Its Treatment. Am. Surg. , 19: 17-23, 1953. JAFFE, H. L.: Osteoid-Osteoma: A Benign Osteoblastic Tumor Composed of Osteoid and Atypical Bone. Arch. Surg. , 31: 709-728, 1935. JAFFE, H. L.: Tumors and Tumorous Conditions of the Bones and Joints, pp. 100-102. Philadelphia, Lea and Febiger, 1958. ROSBOROUGH, D. : Osteoid Osteoma. Report of a Lesion in the Terminal Phalanx of a Finger. J. Bone and Joint Surg. , 48-B: 485-487, Aug. 1966. SANKARAN, BALU: Osteoid Osteoma. Surg. , Gynec. and Obstet., 99: 193-198, 1954. SCHAJOWICZ, FRITz; AIELLO, C. L. ; and SLULLITEL, ISIDoR0: Cystic and Pseudocystic Lesions of the Terminal Phalanx with Special Reference to Epidermoid Cysts. Clin. Orthop. , 68: 84-92, 1970. SEVITT, S., and HORN, J. S.: A Painless and Calcified Osteoid Osteoma of the Little Finger. J. Path. Bacteriol. , 67: 571-574, 1954. SHERMAN, M. S.: Osteoid Osteoma. Review of the Literature and Report of Thirty Cases. J. Bone and Joint Surg. , 29: 918-930, Oct. 1947.

African J. Surg., 5: 13-24, A Report on 80 Cases. Am.

1967. J. Roentgenol.

194-205,

3. 4. 5. 6.
7.

8. 9. 10. 11.
12.

Gangrene
A
BY ROBERT N. From HENSINGER, the Alfred

of the Newborn
CASE REPORT WILMINGTON, Institute, Wilmington DELAWARE M.D.*, I. duPont

than did life

Gangrene of the newborn is uncommon, sixty cases reported 18, in only twenty-four the condition and and
1,3,4,69,14,16,18

with fewer of which day the of exwhose

Her

Case
The mother had was been a gravida controlled II, disease

Report
para I, twenty-six-year-old and died diabetic shortly

develop The lesion

within usually

the first involves

with insulin

since the age of eighteen.

tremities the acute Yet, orthopaedic precipitated end result careful insult
*

orthopaedic reconstructive

consultation management previously

is often required in of these children. discussed in the be the A the as a

Medical

after

first child had been birth from hyaline Six weeks prior to assess without

born prematurely (3,284 grams) membrane disease and atelectasis. date of delivery, fetal lung maturity. The procedure complication. Lethicin/sphingomyelin
that the fetal

to the estimated

an amniocentesis went smoothly ratio #{176} and Five days within not on was the

the condition

has not been

was performed and seemingly stains later to the

literature. by a wide is diminished

Gangrene of the newborn may variety of situations; however, perfusion and local ischemia. of events surrounding the etiology and
University of Michigan

for fat cells the membranes hospital. hours. it was felt and Her

indicated

lungs and

were the

immature. stopped

spontaneously She remained

ruptured, afebrile

and the mother leakage

was admitted

analysis is helpful
Section of

of the sequence in determining

Orthopaedic Surgery,

twenty-four

mature,
antibiotics induced.

As it was believed that advisable to delay delivery. Fourteen felt and was that potential now days the of fetal after

the babys lungs were The mother was placed amniocentesis, to the child the outweighed distress. labor from

discharged. physician

consequence
Center,
VOL.

the treatment
Arbor, NO. Michigan

of the lesion.
48104.
1975

hazards

ruptured
prematurity.

membranes
There

infection

risks

of

Ann 57-A,

no evidence

1, JANUARY

122

ROBERT

N.

HENSINGER

FIG.

1-A

FIG.

1-B

FIG. 2

FIG. 3

Fig. 1-A: Clinical photograph of the patient six hours after delivery. The left upper extremity is edematous from the mid-part of the arm distally. Skin changes on the extensor surface of the forearm appear to be long-standing. The three dark spots were reported to be so-called blood blisters, which broke spontaneously at the time of delivery. Fig. 1-B: Another view of the arm, demonstrating the superficial erosion at the elbow flexion crease and wrist. Fig. 2: Forty-eight hours after delivery the photograph demonstrates the extent of the dry gangrene of the forearm and at the wrist. Three weeks after delivery the eschar sloughed, revealing healthy granulation tissue. Fig. 3: Appearance of the left upper extremity when the patient was fourteen months old. The residual scarring in the elbow flexion crease and on the extensor surface of the forearm does not interfere with elbow function. Note that the extremity is thin from the mid-part of the arm to the wrist. The infant offspring of the distal blue amniotic cord. left labor (Figs. arm (ten 3,284 I-A hours)
grams,

and cephalic
and had

delivery
the

were

uncomplicated. of the mid-portion I-A and were which by the 1-B) three

The

flexion joints sensation

due had was

to contracture range normal.

of

the

wrist

extensors. groups

The were

remainder functioning,

of the and

weighed

typical

appearance (Figs.

of a diabetic and dark broke umbilical except

a full

of motion.

All muscle

and

1-B).

At birth

the appearance was cyanotic. over the

suggested blood during or that

previous extremity blisters

constriction noted There had

to this area the entire so-called band Within

There forearm

Discussion It is important from necrotizing to distinguish fasciitis of the gangrene newborn, of the newborn a much more

spontaneously

the delivery. the extremity the extremity

was no evidence been encircled to a normal

of a congenital color

an hour,

had returned

for an area of whitish (Figs. I-A and 1-B), thermal (Figs. elbow groove palpation, pulse sensation
was noted;

discoloration on the extensor surface of the forearm similar in appearance to the early changes following a six erosion entire hours ofthe extremity The flexor of infant as after skin delivery demonstrated of the to the to A radial forearm, extremity but of all erosion. distal

injury. Examination 1-A and 1-B) and at the similar palpable. was elbow grossly or wrist. were
however,

virulent condition with rapidly developing sepsis and destruction of tissue 19 The latter condition is associated with a high mortality rate and demands an entirely different course including
surgical

at approximately superficial The wrist.

at the flexorcrease compartment

of

treatment prompt and

than vigorous

gangrene supportive

of

the

newborn, and

was edematous Volkmanns the would normal. skin movement move range passive

measures

at the mid-part was

of the arm. With intact. There were cultured included sterile surface of leathery were and weeks) and contracture. the

was tense the

to the findings

following exception the was from interpreted No spontaneous a full

ischemia. over of the

d#{233}bridement 19 Similarly, be proved or the condition develops particularly should be newborn if the part is pale suspected 1,3,68.l6.I8 period include congenital

if direct pressure cannot subsequent to delivery,

if stimulated,

the fingers, of motion skin

and cold, arterial occlusion Other causes later in the heart disease, dehydraor of in the or the the the
and

not joints. and

the

Roentgenograms Enterococcus Initial treatment of the extremity, to prevent the from and extensor forming saline bilirubin of

Enterobacteriaceae

the areas

of superficial

antibiotics,
dressings, Over the

intravenous
elevation, the ensuing

fluids,

a thorough hours, the

tion, sepsis, emboli from a patent ductus arteriosis umbilical vein, venipuncture or cutdown, polycythemia, disseminated intravascular coagulation, or tight binding the legs
1,3,7-9.12.14.15

cleaning exercises skin gradually retracted formed, signs the

and range-of-motion forty-eight frank granulation never necrosis, gradually tissue elevation of

forearm eschar (Fig. The

underwent tissue, infant

a black, soaks sepsis, (three sloughed

2). The eschar

the surrounding

healthy-appearing instituted. with the exception

demonstrated the remainder by secondary but the to be of performed

Gangrene of the newborn seems to be more common pregnancies associated with spontaneous rupture of amniotic sac, particularly if there is a delay in delivery long, difficult, dry labor 1,4.6.7.9.111 In these situations extremity maternal extremity
patient

of generalized hospitalization The eschar with from maintained a soft

of a transient

of the serum

( 16.4 milligrams the

per 100 milliliters) area healed to the extremity splint and quickly

may

prolapse in
the

between

the presenting canal. during

at the

head

and the

was uneventful.

pelvis remains
reported,

the birth compressed

groove

Thus the

trapped, labor. In
of the arm

intention smaller wrist

pliable
the mid-part than in

scar. The induration

of the arm opposite the

gradually
wrist (Fig. the 3). mother

resolved,
Initially,

mid-part

the extremity was

continued

circumference

a cock-up

cyanosis constriction return. color etiology. The were

of

the extremity in the birth canal

noted and

at birth obstruction

suggested of venous of the

range-of-motion exercises relaxed. At fourteen months to use both hands equally.

to the joints. Gradually these measures were the child was healthy and active, and appeared The left wrist was limited to 10 degrees of volar

palpable radial pulse arguments against diabetes may

and quick improvement arterial occlusion as have increased

Maternal

the infants

GANGRENE

OF

THE

NEWBORN

123 and progress However, through the typical stage is

susceptibility risk of venous

to venous thrombosis

occlusion, as there is an increased in newborn offspring of diabetic

allowed of dry alarming reassurance result appearance modified

to demarcate gangrene. to the that in a

the appearance

of the eschar

mothers Valderrama and associates recently reported a case of gangrene of the newborn associated with maternal diabetes. The infant was born with an edematous hand which gradually became cyanotic and eventually gangrenous. The skin changes forearm were of longer The three pinpoint marks noted on the extensor surface of the duration and are difficult to explain. (so-called blood blisters) found on the with the sites. days noted, fetal amniocentesis, However, to delivery, needle marks

parents (and a conservative of

staff as well) and all need approach will ultimately tissue loss. A satisfactory 3) or can be obtained (Fig. procedure.

minimum

of the skin is usually later by a reconstructive

These
decompression

ischemia,

infants are not candidates for surgical of the forearm for the Volkmanns as might be considered for an older child or adult.

the forearm suggest a relationship as they may represent puncture amniocentesis and from days. as Creasman intrauterine Furthermore, was performed and co-workers transfusions it is quite damage Subcutaneous
reported
20,

twelve

prior

Remarkable recovery of muscle function, even after severe ischemia, can occur, as it did in this patient and a similar patient reported by Heller and Alvari, although it may take weeks or months Early range-of-motion are exercises important and in the splinting to avoid joint contractures rehabilitation of the extremity.

generally heal within a few unusual for the fetus to sustain from being struck with infection following amniocenbut have appears become local unlikely entrapped When forearm to the of the a

any permanent needle 2,5,10.20

tesis has been

Summary Gangrene resulting of the newborn from decreased are a variety Knowledge is an uncommon condition perfusion of a part, usually of situations of the exact which sequence can of

patient. time began

The seven

forearm days the later,

may rupture likelihood the

in this at the labor may entire

usually

of membrane

with

ischemia.

tethering

an extremity. There result in this condition. events which lead

have increased extremity.

of constriction

to the insult

and the conditions

surround-

Treatment of this condition adequate hydration, maintenance ment for the extremity, The risk demarcate. antibiotics and has mortality of secondary

is supportive and includes of an aseptic environthe gangrenous infection area to and is high

and allowing

ing it is helpful in determining the quence the treatment of the lesion. direct pressure from the maternal contributing factor which led to
.

etiology and as a conseIn the patient described, pelvis was probably the venous occlusion of the

may be required. Control of infection is essential been credited for significantly reducing the from this condition l#{149} d#{233}bridement or amputation should be reserved
1.3.7.9,14.15

extremity Arterial thrombosis , emboli , trauma, congenital heart disease, sepsis, dehydration, coagulopathies, and venipuncture are other possible causes which should be
considered. The treatment is in general supportive, allowing

Surgical

the is

ischemic

area

to

demarcate

and to avoid

slough. contracture

Range-ofare help-

for the patient in whom gross sepsis develops The area of loss is generally less if the injured

tissue

motion exercises and splinting ful in the rehabilitative phase.

References
1.
2. 3. 4.

5. 6. 7. 8. 9. 10. I 1.
12.

13.
14.

15. 16. 17. 18. 19. 20.

W. E. , and WONG, RUTH: Gangrene of the Extremities in the Newborn Infant. Report of Two Cases. J. Pediat. , 40: 588-598, 1952. BERNER, H. W.; SEISLER, E. P.; and BARLOW, JOHN: Fetal Cardiac Tamponade. A Complication of Amniocentesis. Obstet. and Gynec. , 40: 599-604, 1972. BRALY, B. D. : Neonatal Arterial Thrombosis and Embolism. Surgery, 58: 869-873, 1965. COPECK, GERALD, and TASSY, FRITZ: Gangrene of an Extremity in a Newborn Infant. Arch. Pediat. , 77: 452-456, 1960. CREASMAN, W. T.; LAWRENCE, R. A.; and THIEDE, H. A.: Fetal Complications of Amniocentesis. J. Am. Med. Assn., 204: 949-952, 1968. GILBERT, E. F. ; HOGAN, G. R. ; STEVENSON, M. M. ; and SUZUKI, HIROSHI: Gangrene of an Extremity in the Newborn. Pediatrics, 45: 469-472, 1970. GROSS, R. E.: Arterial Embolism and Thrombosis in Infancy. Am. J. Dis. Child. , 70: 61-73, 1945. HEFFELFINGER, M. J. , and HARRISON, E. G., JR.: Neonatal Gangrene with Developmental Abnormality ofthe Femoropopliteal Artery. Arch. Path., 91: 228-233, 1971. HELLER, GEORGE, and ALvARI, GEORGE: Gangrene of the Extremities of the Newborn. Occurrence of Functional Recovery. Am. J. Dis. Child. , 62: 133-140, 1941. LEMONS, J. A. , and JAFFE, R. B.: Amniotic Fluid Lecithin-Sphingomyelin Ratio in the Diagnosis of Hyaline Membrane Disease. Am. J. Obstet. and Gynec. , 155: 233-237, 1973. LILEY, A. W.: The Technique and Complications of Amniocentesis. New Zealand Med. J. , 59: 581-586, 1960. MANIOS, S. G. ; KANOKOUDI, F. ; and MILIARAS-VLACHAKIS, M.: Gangrene of Lower Extremities in a Newborn Infant Associated with Intravascular Coagulation. (Recession of Gangrene after Heparmn Therapy.) Helvetica Pediat. Acta, 27: 187-192, 1972. OPPENHEIMER, E. H., and ESTERLY, J. R.: Thrombosis in the Newborn: Comparison between Infants of Diabetic and Nondiabetic Mothers. J. Pediat. , 67: 549-556, 1965. PAPAGEORGIOU, APOSTOLOS, and STERN, LEO: Polycythemia and Gangrene of an Extremity in a Newborn Infant. J. Pediat. , 81: 985-987, 1972. PERLMUTTER, H. D. , and WAGNER, D. H.: Arterial Thrombosis in the Newborn Infant. J. Pediat. , 37: 259-262, 1950. STEINER, M. D.: Gangrene of an Extremity in a Newborn Child. Am. J. Obstet. and Gynec. , 49: 686-690, 1945. TORPIN, RICHARD: Fetal Malformations; Caused by Amnion Rupture during Gestation. Springfield, Illinois, Charles C Thomas, 1968. VALDERRAMA, ELSA; GRIBETZ, IRWIN; and STRAUSS, LonE: Peripheral Gangrene in a Newborn Infant Associated with Renal and Adrenal Vein Thrombosis. Report of a Case in an Offspring of a Diabetic Mother. J. Pediat., 80: 101-103, 1972. WEINBERGER, MALVIN; HAYNES, R. E.; and MORSE, T. S.: Necrotizing Fasciitis in a Neonate. Am. J. Dis. Child., 123: 591-594, 1972. WILTCHIK, S. G.; SCHWARZ, R. H.; and EMICH, J. P., JR.: Amniography for Placental Localization. Obstet. and Gynec., 28: 641-645, 1966.
ASKUE,

VOL.

57-A,

NO.

1, JANUARY

1975